Le Infezioni in Medicina, n.

1, 39-41, 2011

Casi
clinici Cystic hydatidosis: a rare case
Case
of spine localization
reports Idatidosi cistica: localizzazione inusuale al rachide

Francesco Scarlata1, Salvatore Giordano2, Laura Saporito1,

Lorenzo Marasà3, Giuseppe Li Pani3, Antonio Odierna Contino4,
Vincenzo Scaglione4, Paola Di Carlo1, Amelia Romano2
1
Dipartimento di Scienze per la Promozione della Salute, Sezione di Malattie Infettive,
Università di Palermo, Palermo, Italy;
2
U.O. Malattie Infettive ARNAS Civico, Palermo, Italy;
3
U.O. Anatomia Patologica ARNAS Civico, Palermo, Italy;
4
U.O. Neurochirurgia ARNAS Civico, Palermo, Italy

n INTRODUCTION known as the PAIR procedure (ultrasonograph-

ic-guided puncture, aspiration of hydatid fluid,

C
ystic hydatidosis is the infection by larvae of
Echinococcus granulosus, a small tapeworm
which requires canines as definitive hosts
and herbivores or humans as intermediate hosts.
Human infection is caused by ingestion of the
tapeworm eggs while playing with infected dogs
(which often present eggs in the fur) or through
consumption of garden vegetables or water con-
taminated by dog feces.
The disease is common in undeveloped areas of
the Mediterranean basin, Middle East, Oceania,
South Africa and South America. Foci also exist
in regions of North America and South Europe.
In Italy the infection is endemic in the Southern
regions and in the isles, mainly in Sardinia.
Liver and lungs are the most common sites of
cyst formation. Less frequently cyst develops in
the kidney, peritoneum, spleen. The involve-
ment of other organs is very infrequent.
Cyst may be symptomless for 10 to 20 years un-
til it becomes large enough to produce symp-
toms. If a cyst ruptures suddenly, a severe al-
lergic reaction or even anaphylaxis and death
may occur.
The diagnosis is most often suspected when ra-
diographic examinations show space-occupy-
ing lesions of internal organs. Serological test-
ing can be helpful despite variable sensitivity.
Surgical removal of cyst is the usual treatment.
Chemotherapy with albendazole is indicated
for the treatment of patients with inoperable
disease or as pre-surgical and post-surgical
treatment to reduce the risk of recurrence [1].
Another intervention for inoperable cysts is
injection of 95% ethanol, re-aspiration) and has
been used successfully at some centers [2].
We present a case of very rare localization of
hydatid cysts in the dorsal spine.
n CASE REPORT
G.C. was a 38-year old housewife admitted to
the Neurosurgery Department at the Civico
Hospital, Palermo, Italy, with lower limbs para-
plegia and urinary retention. The patient had
complained for lower limbs pain for 5 months.
Fever had appeared two weeks before admis-
sion. Her past medical history was remarkable
for pulmonary hydatid cyst disease surgically
treated 25 years before. Six years before she was
diagnosed with spinal hydatid cyst affecting
the VIII thoracic vertebra for which only anal-
gesic treatment was administered. On admis-
sion, the patient presented with flaccid paraple-
gia, bilateral loss of superficial sensation below
the umbilical transversal line and spared deep
sensation. On abdomen examination, a full
bladder, resulting from over-distention, was
noticed. Hematologic and chemical analyses
were within normal ranges. Serological testing
to Echinococcus spp. by enzyme-linked im-
mune-sorbent assay (ELISA) showed high titers
of IgG and IgE specific antibodies.
Computed tomography (CT) of the dorsal and
lumbar spine showed T8 vertebral body col-
lapse and two osteolytic lesions involving T7.
Magnetic resonance imaging (MRI) further-

39
2011
more demonstrated cystic lesions invading the
spinal canal with spinal cord compression and
morphologic alterations also of T9 vertebral
body (Figure 1).
Thoracic CT revealed multiple nodules involv-
ing both pulmonary apices and thickening of
the left parietal and mediastinal pleura with
mediastinal shift to the left side and mediasti-
nal lymph node enlargement.
The patient was started on oral albendazole at
the dose of 400 mg twice daily. One week after,
she underwent T8 laminectomy. Multiple, part-
ly fissured, hydatid cysts causing spinal cord
compression were removed. Ten days after, T7,
T8 and T9 vertebral bodies were excised and re-
placed with titanium prostheses. Histological
examination confirmed the diagnosis of
echinococcosis (Figure 2).
At discharge, the patient presented mild para-
paresis which improved after a program of in-
tensive physiotherapy. Albendazole treatment
Figure 1 - Vertebral MRI. Cystic lesions invading the
spinal canal with spinal cord compression. Collapse
of T8 vertebral body, osteolytic lesions involving T7
and morphologic alterations of T9.
40
2011
Figure 2 - Cuticular membrane and isolated prolige-
rous vesicles in the bone tissue (hematoxylin eosin,
200x).
was maintained until three months after dis-
charge. Until now the patient is on ambulatory
follow up and no relapse had occurred.
n DISCUSSION
Hydatid disease of spine occurs in 1% of all cas-
es of human echinococcosis and is most com-
monly located in dorsal spine (50%) followed
by lumbar (20%), sacral (20%) and cervical
spine (10%). The disease occurs either by direct
extension from pulmonary infestation or rarely
begins primarily in vertebral body. There are
no pathognomonic signs or symptoms. Differ-
ently from other localizations where cyst en-
larges radially, in the bone the parasite grows
multilocularly with dilatation of the spaces of
spongiosa. The cyst breaks out of vertebral
body anteriorly or laterally extending into ex-
tradural spaces or paraspinal tissues and can
extend into the spinal canal compressing the
spinal cord [3].
Symptoms are not specific and are generally re-
lated to spinal cord compression which tend to
cause characteristic radicular syndromes of
pain and segmental neurologic deficits. While
plain radiographic findings are not specific (the
images of tuberculosis, neoplasm or cysts are
similar), MRI is the gold standard for diagnos-
ing hydatid disease of the spine. Hydatid cysts
have inhomogeneous, low signal intensity on
T1 weighted images and hyper intense signal
on T2. The prognosis for hydatid disease of the
spine remains poor. In fact surgical treatment
rarely extirpates completely the cysts. Then the
rate of recurrence is high [4].
The reported case is remarkable for the rarity of
localization and for the absolute need of a mul-
tidisciplinary approach for diagnosing and
monitoring of suspected hydatid lesions. The
positive anamnesis for previous echinococcosis
was helpful for diagnosis.
Indeed, many cases of rare localizations of
echinococcosis remain undiagnosed or misdi-
agnosed for many years with the dangers con-
nected with the possible rupture of the cyst. We
think that patients with either serological posi-
SUMMARY
Cystic hydatidosis is a zoonosis endemic both to
Sicily and other Mediterranean areas. Generally,
Echinococcus granulosus tapeworms develop in the
liver, lung and less frequently in the peritoneum,
spleen or kidney. We present a rare case of spinal
hydatid disease.
The patient was a 38-year-old housewife with a
vertebral echinococcosis revealed by acute para-
plegia of the legs. Medical treatment with albenda-
zole and surgical intervention improved the clini-
RIASSUNTO
L’echinococcosi, o idatidosi, è una zoonosi che costitui-
sce ancora oggi un importante problema sanitario in
numerosi Paesi sia industrializzati che in via di svilup-
po. L’idatidosi è una malattia parassitaria a frequente
localizzazione epatica e polmonare, mentre le altre loca-
lizzazioni sono molto rare anche nelle aree endemiche.
Riportiamo il caso di una paziente di 38 anni affetta da
idatidosi del rachide dorsale che si presentava allo nostra
osservazione per paraplegia acuta degli arti inferiori. Il
trattamento medico con albendazolo e l’intervento neu-
n REFERENCES
[1] Ozdemir M., Diker E., Aydogdu S., Goksel S.
Complete heart block caused by cardiac echinococ-
cosis and successfully treated with albendazole.
Heart 77, 84-85, 1997.
[2] Mawhorter S., Temeck B., Chang R., Pass H.,
Nash T. Nonsurgical therapy for pulmonary hydatid
cyst disease. Chest 112, 1432-1436, 1997.
41
2011
tivity for echinococcosis or diagnostic imaging
findings suggestive for abdominal or lung hy-
datid cysts should be also investigated with
first level methods for other hydatid localiza-
tions as brain, spine, heart. Furthermore in en-
demic areas hydatidosis must be always sus-
pected in presence of occupying space lesions
of these districts.
Key words: Echinococcus granulosus, spinal
echinococcosis.
cal symptoms. This case is emblematic both for the
unusual localization and for the need of a multi-
disciplinary approach for diagnosing and monitor-
ing suspected hydatid lesions.
Patients with suspected abdominal or lung
echinococcosis should also be investigated for oth-
er localizations such as the brain, spine and heart.
Furthermore, in endemic areas hydatidosis must
be suspected in the presence of lesions occupying
space in these districts.
rochirurgico determinarono un miglioramento della sin-
tomatologia. Il caso riportato è emblematico sia per la ra-
rità della localizzazione, sia per l’importanza della ge-
stione pluridisciplinare che ha coinvolto infettivologi,
neurochirurghi ed anatomopatologi. Nel sospetto di ida-
tidosi epatica o polmonare, è necessaria una valutazione
più ampia per individuare eventuali localizzazioni cere-
brali, spinali o cardiache. In aree endemiche, in presenza
di lesione occupante spazio l’eziologia echinococcica deve
essere comunque sospettata anche in sedi non usuali.
[3] Rao S., Parikh S., Kerr R. Echinococcal infestation
of the spine in North America. Clin. Orthop. Relat.
Res. 271, 164-169, 1991.
[4] Schnepper G.D., Johnson W.D. Recurrent
spinal hydatidosis in North America. Case report
and review of the literature. Neurosurg. Focus 17,
E8, 2004.